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Intrathecal baclofen versus selective dorsal rhizotomy for children with cerebral palsy who are nonambulant: a systematic review. 鞘内巴氯芬与选择性背根切断术治疗无行走能力的脑瘫患儿:系统性综述。
IF 2.1 Pub Date : 2019-10-18 Print Date: 2020-01-01 DOI: 10.3171/2019.8.PEDS19282
Benjamin Davidson, Nathan Schoen, Shaina Sedighim, Renée Haldenby, Blythe Dalziel, Sara Breitbart, Darcy Fehlings, Golda Milo-Manson, Unni G Narayanan, James M Drake, George M Ibrahim

Objective: Cerebral palsy (CP) is the most common childhood physical disability. Historically, children with hypertonia who are nonambulatory (Gross Motor Function Classification System [GMFCS] level IV or V) were considered candidates for intrathecal baclofen (ITB) therapy to facilitate care and mitigate discomfort. Selective dorsal rhizotomy (SDR) was often reserved for ambulant children to improve gait. Recently, case series have suggested SDR as an alternative to ITB in selected children functioning at GMFCS level IV/V. The objective for this study was to systematically review the evidence for ITB and SDR in GMFCS level IV or V children.

Methods: Medline, Embase, Web of Science, and Cochrane databases were systematically searched. Articles were screened using the following inclusion criteria: 1) peer-reviewed articles reporting outcomes after SDR or ITB; 2) outcomes reported using a quantifiable scale or standardized outcome measure; 3) patients were < 19 years old at the time of operation; 4) patients had a diagnosis of CP; 5) patients were GMFCS level IV/V or results were reported based on GMFCS status and included some GMFCS level IV/V patients; 6) article and/or abstract in English; and 7) primary indication for surgery was hypertonia. Included studies were assessed with the Risk of Bias in Non-Randomized Studies - of Interventions (ROBINS-I) tool.

Results: Twenty-seven studies met inclusion criteria. The most commonly reported outcomes were spasticity (on the Mean Ashworth Scale) and gross motor function (using the Gross Motor Function Measure), although other outcomes including frequency of orthopedic procedures and complications were also reported. There is evidence from case series that suggests that both ITB and SDR can lower spasticity and improve gross motor function in this nonambulatory population. Complication rates are decidedly higher after ITB due in part to the ongoing risk of device-related complications. The heterogeneity among study design, patient selection, outcome selection, and follow-up periods was extremely high, preventing meta-analysis. There are no comparative studies, and meaningful health-related quality of life outcomes such as care and comfort are lacking. This review is limited by the high risk of bias among included studies. Studies of SDR or ITB that did not clearly describe patients as being GMFCS level IV/V or nonambulatory were excluded.

Conclusions: There is a lack of evidence comparing the outcomes of ITB and SDR in the nonambulatory CP population. This could be overcome with standardized prospective studies using more robust methodology and relevant outcome measures.

目的:脑性瘫痪(CP)是最常见的儿童肢体残疾。一直以来,患有肌张力过高且无法行走(粗大运动功能分级系统 [GMFCS] IV 级或 V 级)的儿童被认为适合接受鞘内巴氯芬(ITB)治疗,以方便护理并减轻不适。选择性背根切断术(SDR)通常被保留给行动不便的儿童,以改善步态。最近,有系列病例表明,选择性背根切断术(SDR)可替代 ITB,适用于功能处于 GMFCS IV/V 级的特定儿童。本研究的目的是系统回顾在GMFCS IV级或V级儿童中使用ITB和SDR的证据:方法:系统检索了 Medline、Embase、Web of Science 和 Cochrane 数据库。文章筛选采用以下纳入标准:1)报道 SDR 或 ITB 术后结果的同行评议文章;2)使用可量化的量表或标准化结果测量方法报告结果;3)手术时患者年龄小于 19 岁;4)患者确诊为 CP;5)患者为 GMFCS IV/V 级,或根据 GMFCS 状态报告结果并包括部分 GMFCS IV/V 级患者;6)文章和/或摘要为英文;7)手术的主要适应症为肌张力亢进。采用非随机干预研究中的偏倚风险(ROBINS-I)工具对纳入的研究进行评估:结果:27 项研究符合纳入标准。最常报告的结果是痉挛(采用平均阿什沃斯量表)和粗大运动功能(采用粗大运动功能测量法),但也报告了包括矫形手术频率和并发症在内的其他结果。系列病例的证据表明,ITB 和 SDR 均可降低痉挛程度,并改善非行动不便人群的粗大运动功能。ITB 术后的并发症发生率明显较高,部分原因是装置相关并发症的持续风险。研究设计、患者选择、结果选择和随访期之间的异质性极高,因此无法进行荟萃分析。没有比较性研究,也缺乏有意义的与健康相关的生活质量结果,如护理和舒适度。本综述因所纳入研究的高偏倚风险而受到限制。未明确描述患者为 GMFCS IV/V 级或不能行走的 SDR 或 ITB 研究被排除在外:结论:目前缺乏证据比较 ITB 和 SDR 在非行动不便的 CP 患者中的疗效。使用更可靠的方法和相关的结果测量指标进行标准化的前瞻性研究可以克服这一问题。
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